Peripheral ameloblastoma is a rare extaosseous odontogenic tumor arising from the tooth-bearing soft tissue of the mandible and maxilla. Fifty-three cases have been recorded so far. Their clinical pictures do not manifest as aggressive and invasive as the central one. Most of them are usually firm, sessile and exophytic. Peripheral ameloblastoma has the similar histologic characteristics of their counter part, mostly benign. Malignant change or distal metastasis is rare. Surgical excision with free safety margin is adequate for the treatment. We reported a rare case of left maxillary sinus tumor with the initial presention of epistaxis. The tumor was removed by left lateral rhinotomy and medial maxillectomy. Microscopic examination revealed peripheral ameloblastome. No bony invasion was found. The post-operative coure was uneventful. No regional recurrence or distal metastasis has been found for 19 months.